Research priorities about stoma‐related quality of life from the perspective of people with a stoma: A pilot survey

Abstract Background There is a recognized need to include patients in setting research priorities. Research priorities identified by people with a stoma are rarely elicited. Objectives To improve the quality of life of people with a stoma through use of evidence‐based practice based on research priorities set by patients. Design and Methods Online pilot survey publicized in 2016 via United Kingdom stoma charities. People ranked nine stoma‐related quality of life topics in order of research priority. Participants People 16 years of age and over who currently have or have had a stoma for treatment for any medical condition. Analysis Distributions of the priority scores for each of the nine research topics were examined. Group differences were explored using either the Mann–Whitney U‐test or the Kruskal–Wallis test depending on the number of groups. Results In total, 225 people completed the survey. The most important research priority was pouch leak problems and stoma bag/appliance problems followed by hernia risk. There were statistically significant differences in ranking research priorities between males and females, age, underlying disease that led to a stoma, stoma type and length of time with a stoma. Conclusion People with a stoma are willing to engage in and set research priorities. The results should contribute towards future research about setting the research agenda for the study of stoma‐related concerns that impact quality of life.


| INTRODUCTION
There are a number of conditions that as part of treatment may necessitate the formation of a stoma including colorectal cancer, diverticular disease, incontinence, ulcerative colitis and Crohn's disease. Each year, for instance, 77% (n≈3000) of people in the United Kingdom undergoing anterior resection for rectal cancer will have a defunctioning or end stoma formed and 27% will still have a stoma at 18-month follow-up. 1 A stoma is an artificial opening in the bowel that has been made to bring the bowel onto the surface of the abdomen in order to divert the flow of faeces or urine. The three types of eliminating stomas are colostomy, ileostomy and urostomy, which can be temporary or permanent. Recent systematic reviews of empirical evidence suggest that a stoma has a negative impact on quality of life of the person with the stoma and that of their spouses. [2][3][4] Hence, identifying factors associated with poor quality of life and recognizing effective solutions are important for people with a stoma. Moreover, stoma-related problems causing the most concern for people with a stoma should arguably take precedence and be research priorities.
In the United Kingdom, there is a recognized need to include patients, carers and clinicians in setting research priorities. 5 Recently, the membership of the Association of Coloproctology of Great Britain and Ireland (ACPGBI) was involved in a Delphi study to reach a consensus on prioritizing clinical research questions in colorectal disease. 6 The study produced a list of 25 research questions that can be considered to reflect the clinical matters of greatest importance by this expert panel. A similar research priority setting exercise was conducted by the American Society of Colon and Rectal Surgeons. 7 The top-scoring ACPGBI non-cancer questions related to prevention and treatment of colorectal complications, including anastomotic leakage, parastomal hernia and the need for a defunctioning stoma. 7 Top-scoring cancerrelated research questions were; "Is there a price to cancer survival after treatment for colon, rectal and anal cancer? What is the impact of treatment on quality of life? What level of poor function is justified to avoid a permanent stoma?" 7 Hence, surgeons clearly perceive stoma and quality of life for people with colorectal disease as national research priorities.
The ACPGBI acknowledge that there is a need to involve patient support groups in developing these research questions and establishing whether the questions resonate with patients as well as clinicians. 7 To address this, we conducted a pilot survey of people with experience of having a stoma to identify research priorities about stoma-related quality of life. The findings of this pilot survey could then be used to inform the conduct of large-scale studies about research priorities for people with a stoma. In conjunction with the research priorities identified by the ACPGBI, this could contribute towards identification of the focus of future grant applications and act as a guide for future investigators and funders. Ultimately, the purpose is to improve the quality of life of people with a stoma through use of evidence-based practice based on research priorities set by patients.

| METHODS
A pilot cross-sectional survey to determine research priorities about stoma-related quality of life was conducted over 3 months in 2016.
A pilot survey is a preliminary survey used to gather information prior to conducting a survey on a larger scale. The survey was closed after this 3-month period. It was administered online using Bristol Survey On-line (BOS), which is an online service that allows researchers to develop, deploy and analyse an online survey (https://www. onlinesurveys.ac.uk). An online questionnaire is a well-established method used in health research 8

| Recruitment
The inclusion criterion was people 16 years of age and over who cur- to the study's online questionnaire. The Colostomy Association, Urostomy Association and Ileostomy Association represent people with a stoma, providing support, advice and practical information and Bowel and Cancer Research is a charity funding research. Only people who indicated consent by ticking a consent box could access the questionnaire, which is an approach to obtaining consent used in previous online health research projects. 9 The questionnaire was in English, and the sampling approach and recruitment procedure were chosen for convenience and are commensurate with the purpose and scale of the pilot study.

| Variables
Single items were used to gather data on respondents' gender, age when first stoma was formed, underlying disease that led to a stoma, type of stoma, if still had a stoma, and length of time with a stoma.
This information was gathered to examine, for example, whether females and males had different research priorities and so on.

| Analysis
The following characteristics of the people who completed the survey were summarized using descriptive statistics: gender, age when first stoma was formed, underlying disease that led to a stoma, type were examined. The Mann-Whitney test was used to explore differences by age (2 groups: <=50 years old, <50 years old) or gender (2 groups: male, female). The Kruskal-Wallis test was used to explore differences by underlying disease (categorized into five groups: bladder cancer, colon cancer, rectal cancer, inflammatory bowel disease, other), type of stoma (3 groups: colostomy, ileostomy, urostomy) or length of time with stoma (4 groups: <=12 months, 13-24 months, 25-48 months and >48 months). The additional research priority topics identified by patients were listed and the proportion of people identifying each topic was calculated.

| Characteristics of people completing the survey
There were 225 people completing the survey (Table 1). One hundred and sixty-four were female (73%). Sixty-nine (30.6%) were 61 years of age and over when they first had a stoma. Two hundred and fourteen

| Gender
There were significant differences in ranking research priorities be-

| Age
There were significant differences in ranking research priorities by age for two research priority topics. Those who were older when they first had a stoma (>50 years) ranked hernia risk as a more important research priority than those who were younger when they first had a stoma (<=50 years) (mean rank 91. 16

| Length of time with a stoma
Kruskal-Wallis analysis showed that there was a significant difference in ranking research priorities by length of time with a stoma for one research topic. People who had a stoma for <=12 months (mean rank 90.68) or for >48 months (mean rank 88.08) ranked pain and feeling uncomfortable as a more important research priority than others (mean rank 120.71 for 13-24 months and 106.60 for 25-48 months).

| Additional topics
Fifty-four percentage (n=121) of people who completed the survey identified additional topics as research priorities. A minority (n=4) listed more than one additional topic. Topics that were identified by ≥5 people completing the survey are presented in Table 3. It is clear that most people who identified further topics focus on the physical problems caused by a stoma such as skin problems and flatulence.
Topics that were identified by ≤5 people completing the survey are as follows: public awareness and perceptions of stoma, information about reversal, impact on family, hernia, exercising, returning to work, early diagnosis and cure, clothing and support garments, pregnancy and weight loss.

| DISCUSSION
This survey produced a ranking of priorities for stoma-related quality of life research of greatest important to people with a stoma.
This is the first attempt to specifically engage people with a stoma to decide on research priorities using an online survey. The ACPGBI recommended the involvement of patients in developing research priorities. 7 This study did this through a United Kingdom pilot survey of people with a stoma. The research priorities identified in this pilot study represent the perspectives of people with a stoma; 95% of those who replied still had a stoma.
The ACPGBI wished to find out if the research questions identified by clinicians would be supported by patients. 7 Our survey suggests that they would be. The ACPGBI highlighted anastomotic leakage, have issues related to leakage and ballooning. 11 There is also strong evidence that peristomal skin complications, which cause leakage, are endemic. 12 The second top research priority identified in this pilot survey was hernia risk. The reported incidence of parastomal herniation is variable, ranging from 10%-52%. 13 The reasons why a colostomy appears twice as likely to herniate as an ileostomy are not fully known beyond possibly the increased trephine diameter. 14 What is known is that parastomal herniation not only causes further change in body image and cosmesis and increased pain but also a difficulty with stoma appliance application resulting in increased risk of pouch leakage which can result in impaired quality of life. 15 Two large trials about surgical procedures to reduce the risk of parastomal herniation are underway in the United Kingdom. 16,17 There have only been a handful of education and abdominal exercise programmes in the post-surgical period designed to prevent and support those with parastomal hernia. 13,18 Our pilot survey found differences in ranking of research priorities by gender, age, underlying disease that led to a stoma, length of time with a stoma and type of stoma. These differences may reflect variation in problems and concerns experienced by different groups of people with a stoma. One study for instance, found that female colorectal survivors with stomas had more specific psychological and social issues than men including diet management, physical activity, sexuality and social support. 19

| Limitations
There is a risk that our list of nine stoma-related quality of life topics missed key topics for people with a stoma and only reflect those issues deemed important by the research team and the collaborators. Kingdom charities. Nevertheless, we did not ask people completing the survey to indicate where they lived, which means we cannot be certain that most were from the United Kingdom. Finally, because it is a survey advertised using social media, we do not know the total population size and are therefore not able to examine whether the study is representative. To address these limitations, any future largescale survey could recruit from bowel cancer consultant patient lists so that we can calculate, for example the completion rate and study representativeness. Given these limitations, it is premature to claim that certain groups of people with a stoma will have different research prioritizations. Further research is therefore needed to replicate the findings in a more diverse population of people with a stoma.

| CONCLUSIONS
This pilot survey suggests that people with a stoma are willing to engage in and set research priorities about stoma-related quality of life. It is anticipated that these results will contribute towards further research about setting the research agenda for the study of stomarelated concerns that impact quality of life. The question remains as to how this information should be disseminated and used by research funding bodies and researchers.